ArticleGenome biology2020
Successful generation of epigenetic disease model mice by targeted demethylation of the epigenome.
Article in Genome biology, 2020. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 41 papers.
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Who cites it
41 citing papers in PubMed, 54 citations in OpenAlex.
- Epigenetic research methods and animal models for intervertebral disc degeneration (Review).Molecular medicine reports · 2026Review
- Identification of A p300-SP1-BRD4 Transcriptional Axis as a Key Driver of AR Hyperactivation in Polycystic Ovarian Syndrome.Advanced science (Weinheim, Baden-Wurttemberg, Germany) · 2026Article
- Epigenetic editing: from concept to clinic.Nature reviews. Drug discovery · 2026Review
- Epigenome editing based treatment: Progresses and challenges.Molecular therapy : the journal of the American Society of Gene Therapy · 2026Review
- Germline epigenome editing identifies H3K9me3 as a mediator of intergenerational DNA methylation recovery in mice.Nature communications · 2025Article
- Vector-free intra-airway in vivo epigenetic editing.Trends in biotechnology · 2025Article
- Epigenome Engineering Using dCas Systems for Biomedical Applications and Biotechnology: Current Achievements, Opportunities and Challenges.International journal of molecular sciences · 2025Review
- Understanding epigenetic regulation in the endometrium - lessons from mouse models with implantation defects.Epigenomics · 2025Review
- DNA methylation shapes the Polycomb landscape during the exit from naive pluripotency.Nature structural & molecular biology · 2025Article
- Cementum regeneration strategies: Insights from development and periodontal microenvironment.Frontiers in cell and developmental biology · 2025Review
- Allele-specific DNA demethylation editing leads to stable upregulation of allele-specific gene expression.iScience · 2024Article
- The impact of the embryonic DNA methylation program on CTCF-mediated genome regulation.Nucleic acids research · 2024Article
- Epigenome editing revealed the role of DNA methylation of T-DMR/CpG island shore on Runx2 transcription.Biochemistry and biophysics reports · 2024Article
- The long non-coding RNA Meg3 mediates imprinted gene expression during stem cell differentiation.Nucleic acids research · 2024Article
- Article
- Designing Epigenome Editors: Considerations of Biochemical and Locus Specificities.Methods in molecular biology (Clifton, N.J.) · 2024Article
- Neuroepigenetic Editing.Methods in molecular biology (Clifton, N.J.) · 2024Review
- Development of Locus-Directed Editing of the Epigenome from Basic Mechanistic Engineering to First Clinical Applications.Methods in molecular biology (Clifton, N.J.) · 2024Article
- Article
- Current Approaches to Epigenetic Therapy.Epigenomes · 2023Review
Corrections and comments
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Authors and funding
8 authors at 2 institutions in 1 country.
Funding
No grant is acknowledged in the PubMed record.
Abstract
backgroundEpigenetic modifications, including DNA methylation, play an important role in gene silencing and genome stability. Consequently, epigenetic dysregulation can cause several diseases, such as cancer, obesity, diabetes, autism, and imprinting disorders.
resultsWe validate three methods for the generation of epigenome-edited mice using the dCas9-SunTag and single-chain variable fragment-TET1 catalytic domain. We generate model mice for Silver-Russell syndrome (SRS), an imprinting disorder, by target-specific DNA demethylation in the H19 differentially methylated region. Like SRS patients, these mice show H19 upregulation and Igf2 downregulation, leading to severe intrauterine and postnatal growth retardation.
conclusionThis is the first report of an imprinting disease model animal generated by targeted demethylation of specific loci of the epigenome in fertilized eggs. Epigenome-edited animals are also useful for exploring the causative epimutations in epigenetic diseases.
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Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.